Outcome of Monochorionic Diamniotic Twins with Twin Reversed Arterial Perfusion Sequence: Interstitial Laser versus Endoscopic Cord Occlusion.

Twin reversed arterial perfusion sequence (TRAP) is a rare complication of monochorionic twins (MC). This study aimed to describe and compare the short- and long-term outcomes of MC pregnancies with the TRAP sequence treated with two different techniques: interstitial fetal laser (IFL) (n = 22) versus endoscopic cord occlusion (CO) (n = 24). The study population included 46 MC pregnancies with TRAP. Pregnancy loss within 2 weeks after the procedure occurred in 27% of cases (6/22) in the group treated with IFL and in 8% of cases (2/24) in the group treated with CO. The survival rate of the pump twin was 73% (16/22) in the IFL group and 83% (20/24) in the group treated with CO. The median gestational age at birth was 38 weeks in the group treated with IFL and 35 weeks in the group treated with CO. The rate of preterm birth before 34 weeks was 12.5% (2/16) in the group treated with IFL and 32% (7/22) in the group treated with CO. In the group treated with IFL, there were no cases of neurological disabilities reported by the parents compared to three cases in the CO group. IFL is associated with a higher risk of early pregnancy loss; however, if the pregnancy progresses, it is associated with lower risks of preterm birth and neurological disabilities in the survivors.

Amniocentesis in Twin Pregnancies: Risk Factors of Fetal Loss.

This study aims to determine if second trimester amniocentesis in twin pregnancies provides a significant independent contribution in the prediction of miscarriage or fetal loss at any stage of pregnancy. This was a retrospective cohort study of women with twin gestations booked for routine prenatal care in four fetal medicine units in Poland in the years 2010-2020. The study population included: (1) twin pregnancies that underwent amniocentesis at 16-20 weeks' gestation; (2) twin pregnancies that did not require any further testing and were followed-up routinely. Univariable and multivariable regression analysis was used to define which maternal and pregnancy characteristics provided a significant independent contribution in the prediction of miscarriage and fetal loss at any stage of pregnancy. In the study period, 2645 twin pregnancies were eligible for analysis. There were 144 cases of miscarriage defined as fetal loss of one or both twins before 24 weeks and 40 cases of intrauterine death of one or both twins after 24 weeks. A total number of 162 twin pregnancies underwent amniocentesis at 16-20 weeks' gestation. The rate of miscarriage before 24 weeks and the rate of fetal loss at any stage of pregnancy in the group that underwent amniocentesis was 10.49% and 13.58%, respectively, compared to 5.11% and 6.52% that did not undergo amniocentesis. Multivariable regression analysis showed that factors providing a significant independent contribution in the prediction of miscarriage and fetal loss at any stage of pregnancy were monochorionicity (MC), large intertwin discordance in crown-rump length (CRL), low Pregnancy Related Plasma Protein (PAPP-A) MoM and nuchal translucency (NT) above 95th centile. Amniocentesis in twin pregnancies does not provide a significant contribution in the prediction of miscarriage or fetal loss at any stage of pregnancy.

Management Options for Fetal Bronchopulmonary Sequestration.

To evaluate the prenatal course and perinatal outcome of fetuses with bronchopulmonary sequestration (BPS) managed expectantly or using minimally invasive methods. This was a retrospective study of 29 fetuses with suspected BPS managed between 2010 and 2021 in three fetal medicine centers in Poland. Medline was searched to identify cases of BPS managed expectantly or through minimally-invasive methods. In 16 fetuses with BPS, there was no evidence of cardiac compromise. These fetuses were managed expectantly. Thirteen hydropic fetuses with BPS qualified for intrauterine intervention: a thoraco-amniotic shunt (TAS) was inserted in five fetuses, laser coagulation of the feeding vessel was performed in seven cases, and one fetus had combined treatment. In the combined data from the previous and the current study of various percutaneous interventions for BPS associated with hydrops, the survival rate was 91.2% (31/34) for TAS, 98.1% (53/54) for laser coagulation, and 75% (3/4) for intratumor injection of sclerosant. After taking into account cases with available data, the rate of preterm birth before 37 weeks in the group treated with laser coagulation was 14.3% (7/49) compared to 84.6% (22/26) in the group treated with TAS. The need for postnatal sequestrectomy was lower in the group of fetuses treated with laser coagulation 23.5% (12/51) in comparison to fetuses treated with TAS 84% (21/26). In fetuses with BPS without hydrops, progression of the lesion's volume, leading to cardiac compromise, is unlikely. In hydropic fetuses with BPS, intrauterine therapy using minimally invasive methods prevents fetal demise. Both, the rate of preterm birth and the need for postnatal surgery is significantly lower in the group treated with laser coagulation compared to the group treated with TAS.

Percutaneous Intratumor Laser Ablation for Fetal Sacrococcygeal Teratoma.

OBJECTIVE: To evaluate the efficiency of percutaneous intratumor laser ablation for fetal solid sacrococcygeal teratoma (SCT). SUBJECTS AND METHODS: We carried out percutaneous ultrasound-guided intratumor laser ablation through a 17-gauge needle using an output of 40 W in 7 fetuses with large solid SCT and reviewed the literature for minimally invasive therapy for this condition. RESULTS: Laser ablation was carried out at a median gestational age of 20 (range 19-23) weeks, and in all cases there was elimination of obvious vascularization within the tumor and improvement in cardiac function. Three (43%) babies survived and had surgical excision of the tumor within 2 days of birth, 3 liveborn babies died within 5 days of birth and before surgery, and 1 fetus died within 2 weeks after the procedure. In previous series of various percutaneous interventions for predominantly solid SCT the survival rate was 33% (2/6) (95% CI 9.7-70%) for endoscopic laser to superficial vessels, 57% (4/7) (95% CI 25-84%) for intratumor laser, 67% (8/12) (95% CI 39-86%) for intratumor radiofrequency ablation, and 20% (1/5) (95% CI 3.6-62%) for intratumor injection of alcohol. CONCLUSIONS: In solid SCT, the reported survival from intratumor laser or radiofrequency ablation is about 50%, but survival does not mean success, and it remains uncertain whether such interventions are beneficial or not because the number of fetuses is small and there were no controls that were managed expectantly.

Neonatal survival and kidney function after prenatal interventions for obstructive uropathies.

OBJECTIVES: Prenatal interventions in LUTO (lower urinary tract obstruction) usually are still question of a debate between gynaecologist and paediatric nephrologist. We aimed the study to assess the early survival rate and renal outcome in LUTO foetuses. MATERIAL AND METHODS: The study was a prospective data analysis of 39 foetuses from singleton pregnancies. All pregnant women with LUTO in the foetus were qualified for VAS based on a local practice. The mean time of first urine analysis ranged between 13-30 weeks of pregnancy. Primary end-point analysis included live birth, 28d-survival, pulmonary and renal function assessment in neonatal period. RESULTS: From initial number of 39, six patients miscarried before the procedure was performed. Overall, 33 VAS were performer at the mean 21 week of pregnancy (range 14-30 weeks). 25/39 foetuses survived until delivery. Three neonates died in first 3 days of life. In the first month 3 children required peritoneal dialysis, but at 28 day all children were dialysis-free. Overall survival rate at 28 day was 56%. Renal function preservation of the initial group (39) turned out to be low - 18% (7/39). CONCLUSIONS: Our study showed average survival curves and complications. LUTO in the foetus had mostly unfavourable outcome in the neonatal period. The prenatal intervention did not increase it significantly and did not guarantee the preservation of normal kidney function.

Ventriculo-amniotic shunting for severe fetal ventriculomegaly.

INTRODUCTION: Severe fetal cerebral ventriculomegaly, observed in about 1 in 1000 newborns, is associated with a high risk of perinatal death and neurodevelopmental delay in survivors. The objective of this study was to evaluate the efficiency of ventriculo-amniotic shunting for drainage of severe fetal cerebral ventriculomegaly and the neurodevelopment of survivors at the age of 2 years. MATERIAL AND METHODS: This was a retrospective study of 44 fetuses with severe bilateral ventriculomegaly treated with ventriculo-amniotic shunting in a tertiary fetal therapy center between 2010 and 2015. RESULTS: Shunt insertion was successfully carried out at a median gestational age of 25 weeks (range 20-33 weeks). There were three fetal deaths within 24 hours of the procedure and 41 live births at a median gestational age of 37 weeks (range 28-39 weeks). Neurodevelopment at 2 years of age was evaluated using the Bayley scale in the 38 survivors. In the 27 cases with isolated ventriculomegaly 19 (70.4%; 95% confidence interval [95% CI] 51.5%-84.2%) had normal or mild neurodevelopmental delay and 8 (29.6%; 95% CI 15.6%-48.5%) were moderately or severely delayed. In the 11 with non-isolated ventriculomegaly 2 (18.2%; 95% CI 5.1%-4.8%) had normal or mild neurodevelopmental delay and 9 (81.8%; 95% CI 52.3%-94.9%) babies were moderately or severely delayed. CONCLUSIONS: Ventriculo-amniotic shunting is an option for the management of severe ventriculomegaly and results in normalization of the ventricular diameter. However, a high proportion of survivors have neurodevelopmental delay and the possible beneficial effect of ventriculo-amniotic shunting needs to be assessed by randomized studies.

Combined screening for early and late pre-eclampsia and intrauterine growth restriction by maternal history, uterine artery Doppler, mean arterial pressure and biochemical markers.

BACKGROUND: Pre-eclampsia is a systemic disease connected with high maternal and fetal morbidity and mortality. Despite significant progress achieved in perinatal medicine, pre-eclampsia is still one of the most significant current problems in obstetrics. OBJECTIVES: The aim of the study was to establish diagnostic algorithms for early and late pre-eclampsia (PE) and intrauterine growth restriction (IUGR). MATERIAL AND METHODS: A total of 320 pregnant women between 11 + 0 and 13 + 6 weeks of gestation were recruited for a case-control study. The study group consisted of 22 patients with early PE, 29 patients with late PE and 269 unaffected controls. The following parameters were recorded: maternal history, mean arterial pressure (MAP), mean uterine artery pulsatility index (UtA-PI), and the concentrations of placental growth factor (PlGF), pregnancy-associated plasma protein A (PAPP-A) and free beta-human chorionic gonadotropin (free ß-hCG). RESULTS: A multivariable stepwise logistic regression analysis indicated that the best screening model for the prediction of early PE is based on a combined analysis of maternal risk factors, UtA-PI and PlGF levels (sensitivity: 91%; specificity: 84%). The best screening model for the prediction of late PE is based on a combined analysis of maternal risk factors, UtA-PI and MAP (sensitivity: 85%; specificity: 83%). The most effective screening model for the prediction of IUGR is based on a combined analysis of maternal risk factors, UtA-PI and PlGF concentrations (sensitivity: 91%; specificity: 83%). CONCLUSIONS: The integrated model of screening established in this study can be a valuable method to identify patients at increased risk of developing pre-eclampsia and related complications. The ability to predict the occurrence of pre-eclampsia in early pregnancy would enable maternal and fetal morbidity to be reduced through the introduction of strict obstetric surveillance as well as planned delivery in a reference center.

Thoracoamniotic Shunts in Macrocystic Lung Lesions: Case Series and Review of the Literature.

OBJECTIVE: To evaluate the efficiency of thoracoamniotic shunts for drainage of macrocystic-type congenital cystic adenomatoid malformation (CCAM). SUBJECTS AND METHODS: This was a retrospective study of 12 fetuses with a large thoracic cyst treated with thoracoamniotic shunting between 2004 and 2014 in a tertiary fetal therapy center. Medline was searched to identify cases of CCAM treated with thoracoamniotic shunting. RESULTS: In all cases the thoracic cyst was associated with major mediastinal shift, the CCAM volume ratio (CVR) was >1.6, and in eight cases there was associated hydrops. Shunt insertion was successfully carried out in all cases at a median gestational age of 24 weeks (range 18-34). In 10 cases there was live birth at a median age of 38 weeks (range 35-41), but in two hydropic fetuses there was intrauterine death. A literature search identified a total of 98 fetuses with CCAM treated with thoracoamniotic shunting between 1987 and 2016. In the combined data from the previous and the current study, the survival rate was 77% (53 of 69) for hydropic and 90% (37 of 41) for nonhydropic fetuses. CONCLUSIONS: The role of thoracoamniotic shunting in macrocystic lung lesions associated with hydrops is well accepted. Intrauterine intervention is also likely to be beneficial in the subgroup of nonhydropic fetuses with a CVR >1.6.

Biochemical markers in screening for preeclampsia and intrauterine growth restriction.

OBJECTIVES: The aim of the study was to evaluate the relationship between the concentrations of substances released by the placenta: placental growth factor (PIGF), pregnancy-associated plasma protein A (PAPP-A) and free beta-human chorionic gonadotropin (beta-hCG) and the risk of early and late preeclampsia (PE) and intrauterine fetal growth restriction (IUGR). MATERIAL AND METHODS: A total of 180 pregnant women between 11+0 and 13+6 weeks gestation were recruited for a case-control study Twenty-two patients suffered from early PE, 29 patients from late PE. Data analyzed during the study included maternal histoty and concentrations of PAPP-A, PlGF beta-hCG. RESULTS: The multiple of the median (MoM) value of the PAPP-A concentrations was 1.01 in the control group (interquartile range (1QR), 0.65-1.55), 0.67 (1QR, 0.382-0.82) in the group of patients with early preeclampsia and 0.74 (IQ, 0.33-1.09) in the group of patients suffering from late preeclampsia. MoM value of the PIGF concentrations was 1.21 in the control group (IQR, 0.93-1.57), 0.62 (IQR, 0.51-0.96) in the group of patients with early preeclampsia and 0.92 (lQR, 0.63-1.09) in the group of patients suffering from late preeclampsia. MoM value of beta-hCG concentrations was 1.14 in the control group (IQR, 0.75-1.49), 1.08(IQR, 0.74-1.23) in the group of patients with early preeclampsia and 1,25(IQR, 1,05-1,49) in the group of patients suffering from late preeclampsia. The performance of screening was determined by the areas under the curve and detection rates, with a fixed false-positive rate of 10%. CONCLUSIONS: Decreased levels of PAPP-A and PIGF are related to an increased risk of preeclampsia and its complications.

[Fetal therapy--evaluation of intrauterine therapy in congenital cystic adenomatoid malformation of the lung (CCAM)]. / Terapia plodu--ocena skutecznosci leczenia wewnatrzmacicznego wrodzonego zwyrodnienia gruczolakowato-torbielowatego pluc (CCAM).

OBJECTIVES: The aim of the study was to evaluate the efficiency of intrauterine treatment of large cysts in fetal lungs using thoracoamniotic shunts. MATERIAL AND METHODS: Our observational retrospective study was carried out on a series of 8 fetuses who under went thoracoamniotic shunting after sonographic statement of large macrocystic lesions in the lungs at the Department of Gynecology Fertility and Therapy of the Fetus, Polish Mother's Research Institute, between 2009-2014. RESULTS: Mean gestational age at shunt insertion was 26.6 (range 18-33) weeks. Marked mediastinal shift in the echocardiographic examination was observed in all of the investigated cases. Five fetuses had polyhydramion, with 4 hydropic cases. Out of the remaining 4 fetuses without impaired cardiac function, 3 had very large lesions at initial presentation and 1 had a lesion that was rapidly increasing in size. Shunt insertion was successful in all cases. Only one patient went into premature labor (at 36 weeks of gestation). Mean gestational age at delivery was 38.2 weeks. Cesarean section was necessary in the half of the patients due to obstetric complications. All newborns underwent resection of the lesions. Three of them were operated in the first month after birth. The rest of the operations were postponed. Prenatal diagnosis of congenital cystic adenomatoid malformations was confirmed by pathologists in all cases. CONCLUSION: Intrauterine therapy of macrocystic lesions in fetal lungs enables to achieve good perinatal outcome It needs to be considered in every case of a fetus with developing impaired cardiac function.

[Fetal therapy--evaluation of ventriculo-amniotic shunts in the treatment of hydrocephalus]. / Terapia plodu--ocena zastosowania shuntu komorowo-owodniowego w leczeniu wodoglowia.

OBJECTIVE: The aim of the study was to establish optimal diagnostic and therapeutic scheme and to assess the efficacy of intrauterine therapy of hydrocephalus. MATERIAL AND METHODS: The study was carried out between 1992-2012 on the total of 222 fetuses with hydro- cephalus, using Orbis-Sigma and ACCU-Flow valves (168 cases) and Cook8 shunts, according to a strictly defined diagnostic and therapeutic scheme. RESULTS: In the first stage of the study (between 1992-2001), a total of 168 fetuses with prenatally diagnosed hydrocephalus received intrauterine therapy In 91.6% of the cases the therapy resulted in a decreased size of cerebral ventricles. The valve dislocated in 23 cases (13.6%). Preterm delivery occurred in 44% of the affected neonates. Severe mental impairment occurred in 17.76%, average in 36.8%, and slight in 32.9% of the infants. Normal mental development at the age of 3 was observed in 12.5% of the children. A total of 11.2% of chldren did not require further neurosurgical treatment. In the second stage of the study (between 2006-2012) after therapy the size of the right lateral cerebral ventricle decreased by 54.76% (average of27.54 mm to 12.46 mm) and the left lateral cerebral ventricle decreased by 53.12% (average of 26.41 mm to 12.38 mm) (p=0.0018). The maximum and minimum width of the cerebral cortex increased by 23.06% and 27% (average of 9.04 mm to 11.75 mm vs. 3.65 mm to 5 mm), respectively Early complications were observed in 22% of the cases: PROM (6), intrauterine fetal death (4), intrauterine infection (1), and premature detachment of the placenta (1). Average gestational age at delivery was 34 weeks, and 24% of the patients delivered at term. CONCLUSIONS: Implantation of ventriculoamniotic shunts proved to be an effective form of therapy resulting in normalization of intracranial pressure. In both stages of therapy reduction of ventricular size in patients with hydrocephalus and good neurological outcome (45.4% in I stage, 60% in II stage) were observed. In the second stage of therapy the size of lateral brain ventricles after fetal therapy was significantly lower (54%). A total of 18% of the neonates did not require neurosurgical treatment.

[Successful intrauterine therapy in a fetus with a large lung cyst in the third trimester of pregnancy--case report]. / Skuteczna interwencja wewnqtrzmaciczna u plodu z olbrzymiq torbiela pluca w trzecim trymestrze ciazy--opis przypadku.

Congenital cystic adenomatoid malformation is a rare disorder of the respiratory system which occurs with an incidence of 1/25,000-35,000. Depending on the size of the cysts, CCAM is classified into microcystic and macrocystic. Very large lesions carry a significant risk of causing both, pulmonary hypoplasia due to compression of the lung tissue and fetal hydrops, probably due to impaired cardiac function as a result of mediastinal shift and compression of the vena cava. In this report, we present a case of a fetus with prenatally diagnosed large cyst of the left lung. Despite advanced gestational age, the baby was treated by thoracoamniotic shunting owning to developing impaired cardiac function and polyhydramion. Spectacular effects of the therapy have been achieved.